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 Table of Contents  
CASE REPORT
Year : 2016  |  Volume : 3  |  Issue : 1  |  Page : 74-78

Calcifying epithelial odontogenic tumor of the posterior maxilla


1 Department of Oral Medicine and Radiology, A B Shetty Memorial Institute of Dental Sciences, Mangalore, Karnataka, India
2 Department of Oral Medicine and Radiology, New Horizon Dental College and Research Institute, Bilaspur, Chattisgarh, India
3 Department of Ophthalmology, K S Hegde Medical Academy, Nitte University, Deralakatte, Mangalore, Karnataka, India

Date of Web Publication22-Dec-2015

Correspondence Address:
Vidya Ajila
Department of Oral Medicine and Radiology, A B Shetty Memorial Institute of Dental Sciences, Nitte University, Deralakatte, Mangalore - 575 018, Karnataka
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/2348-3334.172391

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  Abstract 

Calcifying epithelial odontogenic tumor (CEOT) is a rare odontogenic neoplasm comprising <1% of all odontogenic tumors. It is commonly seen in the third to fifth decades of life without any gender predilection. It usually occurs in the mandibular posterior region. A painless, slow growing swelling with bone expansion is the most common clinical feature of CEOT. Radiographically, it presents as a mixed lesion with or without an associated impacted tooth. Confirmation of the diagnosis is by histopathological examination. We describe an unusual case of CEOT occurring in the maxillary posterior region and involving the maxillary sinus. The associated impacted third molar was displaced to the lateral wall of the nose and root resorption was seen in all the teeth associated with the lesion. There was no evidence of calcification in conventional as well as computed tomography images.

Keywords: Calcifying epithelial odontogenic tumor, impacted tooth, maxilla


How to cite this article:
Ajila V, Gopakumar R, Hegde S, Shetty H. Calcifying epithelial odontogenic tumor of the posterior maxilla. CHRISMED J Health Res 2016;3:74-8

How to cite this URL:
Ajila V, Gopakumar R, Hegde S, Shetty H. Calcifying epithelial odontogenic tumor of the posterior maxilla. CHRISMED J Health Res [serial online] 2016 [cited 2020 Sep 24];3:74-8. Available from: http://www.cjhr.org/text.asp?2016/3/1/74/172391


  Introduction Top


Calcifying epithelial odontogenic tumor (CEOT) was first described by the Dutch pathologist Jens Jorgen Pindborg in 1955.[1] In 1963, Shafer and colleagues coined the term “Pindborg's tumor.”[1] CEOT is a rare benign odontogenic neoplasm accounting for approximately 1% of all odontogenic neoplasms.[2],[3]

CEOT commonly occurs in the posterior mandible in the premolar-molar region.[4] There is no gender predilection and it is seen in the third to fifth decade of life. Clinically, it most commonly presents as an asymptomatic, slow-growing swelling of the jaws.[4] The radiographic presentation can vary from pericoronal radiolucency to radiolucent areas with diffuse opacities.[4]

This report describes a case of CEOT in the maxillary posterior region associated with an impacted third molar. Radiographically, the lesion showed complete involvement of the maxillary sinus and displacement of the maxillary third molar to the lateral wall of the nose.


  Case Report Top


A 35-year-old-male patient reported to the Department of Oral Medicine and Radiology with complaint of swelling over the right side of the face since 3 months. The swelling was gradually increasing in size and asymptomatic. Extraoral examination showed a diffuse swelling measuring approximately 3 cm × 4 cm over the middle third of right side of the face. Intraoral examination revealed a well-defined swelling extending from the mesial of maxillary right canine to the distal of right maxillary second molar on the buccal aspect. Obliteration of the buccal vestibule was noted [Figure 1]. Maxillary right third molar was missing. On palpation, the swelling was bony hard in consistency and was nontender. Mucosa over the swelling was normal. The maxillary right first and second molar were nonvital. Based on the history and clinical features, provisional diagnosis of dentigerous cyst involving the maxillary right third molar was given.
Figure 1: Intraoral photograph showing well-defined swelling extending from the mesial of maxillary right canine to the distal of right maxillary second molar on the buccal aspect

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Differential diagnosis of the calcifying epithelial odontogenic cyst, adenomatoid odontogenic tumor, and the keratocystic odontogenic tumor was considered.

The intraoral periapical radiograph showed a well-defined unilocular radiolucency extending from right maxillary first premolar to right second molar. There were no calcifications within the lesion. Root resorption was observed in the right maxillary first and second premolar and first and second molar [Figure 2]. The occlusal radiograph showed buccal cortical expansion. A panoramic radiograph revealed a well-defined unilocular radiolucency measuring approximately 5 cm × 6 cm in size extending from the mesial of maxillary right first premolar to the distal of right maxillary second molar. Thinning of the walls of the right maxillary sinus was seen with an impacted tooth displaced superomedially within the sinus [Figure 3]. Paranasal sinus view [Figure 4] showed opacification of the right maxillary sinus. The sinus walls were expanded and thinned. The impacted maxillary right third molar was displaced to the lateral wall of the nose. To determine the extent of the lesion and involvement of surrounding structures, computed tomography (CT) was done.
Figure 2: Intraoral periapical radiograph showing unilocular radiolucency extending from mesial of right maxillary first premolar to right maxillary second molar with root resorption

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Figure 3: Panoramic radiograph showing unilocular radiolucency extending from mesial of right maxillary first premolar to right maxillary second molar with root resorption. The lesion involves the right maxillary sinus causing thinning of the walls. Impacted maxillary third molar is seen displaced superomedially (black arrow)

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Figure 4: Paranasal sinus view showing opacification of the right maxillary sinus and indistinct sinus walls. Impacted maxillary right third molar is displaced to the lateral wall of the nose

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CT revealed a hypodense lesion involving the entire right maxillary sinus. Thinning and perforation of the lateral wall of the sinus was noted. An impacted tooth was seen displaced against the lateral wall of the nose. No calcifications were appreciated in the CT [Figure 5]. Surgical excision of the lesion was done [Figure 6] and histopathology showed connective tissue capsule and epithelial lining with tumor mass. Sheets of polyhedral epithelial cells were present in a connective tissue stroma. Cells were closely packed with distinct cellular outline, eosinophilic cytoplasm, prominent intercellular bridges and slight nuclear pleomorphism. Areas of amyloid like material and basophilic areas of calcification were also noted [Figure 7] Based on the histopathological features, a final diagnosis of calcifying epithelial odontogenic tumor was made.
Figure 5: Axial (a and b) and coronal (c and d) computed tomography showing a hypodense lesion in the right maxillary sinus causing thinning and perforation of the lateral wall of the sinus. No evidence of calcification. An impacted tooth is displaced to the lateral wall of the nose

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Figure 6: Excised surgical specimen showing the lesion along with the associated teeth

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Figure 7: Pictomicrograph showing sheets of epithelial cells, amyloid and calcified material (a) ×10, (b) ×40

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  Discussion Top


In 1992, the World Health Organization classified CEOT as a benign odontogenic tumor which is exclusively of epithelial origin.[5] It has been described previously as ameloblastoma of unusual type with calcification, calcifying ameloblastoma, malignant odontoma, cystic complex odontoma, and variant of simple ameloblastoma.[6] CEOT can be intraosseous or extraosseous in origin.[7] Intraosseous CEOT is the major type occurring in 85% of cases, whereas the extraosseous type occurs in around 6% of cases.[7]

The exact origin of the tumor is unknown. It is believed to arise from the stratum intermedium of enamel organ since the tumor cells appear similar to cells of the stratum intermedium and have high activity of alkaline phosphatase and adenosine triphosphate.[5] Also, the amyloid in CEOT is said to be an immunological response to cells of the stratum intermedium. The extraosseous type of CEOT arises from dental lamina epithelial rests or the basal cells of gingival epithelium.[7]

CEOT commonly occurs in the third to fifth decade of life with a mean age of 40 years.[4] Our patient was in the fourth decade of life. There is no gender predilection.[8] CEOT is twice as common in the mandible.[8] Two-thirds of cases affect the mandibular posterior region.[8] We are reporting a case of CEOT in the posterior maxilla. About 50% of cases of CEOT are associated with an impacted tooth or odontome.[8] The present case was associated with an impacted third molar. Extraosseous CEOT commonly affects the anterior gingiva.[9]

Clinical features include a painless slow growing lesion causing bone expansion.[6] Maxillary lesions may cause symptoms such as epistaxis, nasal stuffiness and headache.[6] Even though the impacted tooth was displaced to the lateral wall of the nose and maxillary sinus was completely involved, our patient did not report any history of epistaxis, nasal stuffiness or headache.

A variety of radiographic features has been described in CEOT, which include pericoronal or nontooth related radiolucency, mixed radiolucent-radiopaque or dense radiopacity, unilocular or multilocular lesions with well-defined or diffuse borders.[10] Kaplan et al.[10] in their study found that the mixed radiolucent-radiopaque appearance was the most common followed by the radiolucent type. The radiopaque type and the classical snow driven appearance described in the literature are rare.[10] In the mixed type with impacted tooth, coronal clustering of the radiopaque material is usually seen.[6] Most reports mention that smaller lesions are radiolucent, whereas the larger ones show mixed radiolucent-radiopaque appearance.[6] This suggests that the duration and size of the tumor may affect its radiographic appearance. However, our case was completely radiolucent regardless of the large size of the lesion. Root resorption has been reported to occur in only 4% of cases.[10] Our case showed root resorption in all the teeth associated with the lesion. CT is used to obtain additional information regarding cortical perforation, evidence of calcifications, location of the impacted tooth and also the extension and proximity of the tumor to surrounding structures.[6] CT images are important for surgical planning and management. In our case, CT was helpful in determining the extension of the tumor and displacement of the tooth. Kaplan et al.[10] mention that dentigerous cysts commonly occur around third molar teeth, whereas CEOT usually involves the first or second molar. However, in our case, CEOT was associated with an impacted third molar.

The differential diagnosis of CEOT includes calcifying epithelial odontogenic cyst, adenomatoid odontogenic tumor, ameloblastoma, ameloblastic fibroodontoma, odontogenic fibroma and ossifying fibroma.[9] In our case, we considered dentigerous cyst in the differential diagnosis due the presence of impacted third molar, unicortical bone expansion, root resorption and absence of calcifications radiographically. Unicystic ameloblastoma can cause significant root resorption and was included in the differential diagnosis. The keratocystic odontogenic tumor can occur with an impacted tooth but do not cause root resorption. The adenomatoid odontogenic tumor occurs in the anterior region of the jaws, may be associated with an impacted tooth, but shows radiopacities and does not cause root resorption. A calcifying epithelial odontogenic cyst is a radiolucent lesion, may be associated with an impacted tooth, but usually has calcified areas radiographically.

The diagnosis of CEOT is by histopathological examination. CEOT is characterized histopathologically by epithelial cells, amyloid-like material, and calcifications. The polygonal epithelial cells can be arranged in nests, sheets or in the cribriform or pseudoglandular pattern. They contain clear to eosinophilic cytoplasm, vesicular nuclei with prominent nucleoli and moderate pleomorphism.[7] Necrosis and atypical mitosis are rare. Eosinophilic material resembling amyloid is characteristic of CEOT. Calcifications can vary from few small round areas to Liesegang rings and large aggregates.[7] The Langerhans cell variant shows very small islands and cords of neoplastic cells with abundant amyloid substance and no calcification.[3] A clear cell variant is also described in the literature.[11] Our case showed sheets of polyhedral epithelial cells with a distinct cellular outline, eosinophilic cytoplasm, prominent intercellular bridges and slight nuclear pleomorphism. Areas of amyloid-like material and basophilic areas of calcification were also present.

The presence of calcifications in CEOT may have prognostic implications. The absence of calcification indicates a poorly differentiated tumor with more chance of recurrence.[7]

The treatment plan is dependent on the size of the tumor, the location of the tumor, general condition of patient and operator skill. Small, well-defined, intrabony mandibular lesions can be treated by simple enucleation or curettage and removal of a thin layer of bone next to the tumor.[5] Larger tumors may need segmental resection, hemimandibulectomy and hemimaxillectomy, followed by bone grafting or distraction osteogenesis.[5] In our case, surgical excision along with the associated teeth was done.

The recurrence rate varies from 10% to 20% with the clear cell variant having a higher recurrence rate of around 15%.[5],[8] Malignant transformation is rare.[8]

Thus, our report describes a case of CEOT with atypical clinical and radiographic features, which include occurrence in the posterior maxilla, complete involvement of the maxillary sinus, displacement of impacted third molar to the lateral wall of the nose, root resorption of all teeth associated with the lesion and absence of calcifications radiographically.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.

 
  References Top

1.
Akhtar K, Khan N, Zaheer S, Sherwani RK, Abrar S. Pindborg tumor in an adolescent-An atypical presentation. J K Sci 2009;11:146-7.  Back to cited text no. 1
    
2.
Pereira OH, de Carvalho LP, Brasileiro VL Jr, de Figueiredo CR. Calcifying epithelial odontogenic tumor. Case Rep Pathol 2013;2013:725380.  Back to cited text no. 2
    
3.
Chen Y, Wang TT, Gao Y, Li TJ. A clinicopathologic study on calcifying epithelial odontogenic tumor: With special reference to Langerhans cell variant. Diagn Pathol 2014;9:37.  Back to cited text no. 3
    
4.
Ching AS, Pak MW, Kew J, Metreweli C. CT and MR imaging appearances of an extraosseous calcifying epithelial odontogenic tumor (Pindborg tumor). AJNR Am J Neuroradiol 2000;21:343-5.  Back to cited text no. 4
    
5.
Singh N, Sahai S, Singh S, Singh S. Calcifying epithelial odontogenic tumor (Pindborg tumor). Natl J Maxillofac Surg 2011;2:225-7.  Back to cited text no. 5
[PUBMED]  Medknow Journal  
6.
Deboni MC, Naclério-Homem Mda G, Pinto Jr DS, Traina AA, Cavalcanti MG. Clinical, radiological and histological features of calcifying epithelial odontogenic tumor: Case report. Braz Dent J 2006;17:171-4.  Back to cited text no. 6
    
7.
Kaushal S, Mathur SR, Vijay M, Rustagi A. Calcifying epithelial odontogenic tumor (Pindborg tumor) without calcification: A rare entity. J Oral Maxillofac Pathol 2012;16:110-2.  Back to cited text no. 7
[PUBMED]  Medknow Journal  
8.
Müller D, Manojlovic S, Luksic I, Grgurevic J. Calcifying epithelial odontogenic tumor of the maxilla (Pindborg tumor). Coll Antropol 2012;36:205-8.  Back to cited text no. 8
    
9.
Krishna KS, Vinod VC, Venkateshwarlu M, Sridevi K, Reddy GS. Pindborg tumour: Review of literature and case reports. JIAOMR 2011;23:660-3.  Back to cited text no. 9
    
10.
Kaplan I, Buchner A, Calderon S, Kaffe I. Radiological and clinical features of calcifying epithelial odontogenic tumour. Dentomaxillofac Radiol 2001;30:22-8.  Back to cited text no. 10
    
11.
Habibi A, Saghravanian N, Zare R, Jafarzadeh H. Clear cell variant of extraosseous calcifying epithelial odontogenic tumor: A case report. J Oral Sci 2009;51:485-8.  Back to cited text no. 11
    


    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5], [Figure 6], [Figure 7]



 

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